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Feasibility of Uterine Preservation in the Management of Early-Stage Uterine Adenosarcomas: a Single Institute Experience

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Feasibility of Uterine Preservation in the Management of Early-Stage Uterine Adenosarcomas: a Single Institute Experience Lee et al. World Journal of Surgical Oncology (2017) 15:87 DOI 10.1186/s12957-017-1137-0 RESEARCH Open Access Feasibility of uterine preservation in the management of early-stage uterine adenosarcomas: a single institute experience Young-Jae Lee, Dae-Yeon Kim*, Dae-Shik Suh, Jong-Hyeok Kim, Yong-Man Kim, Young-Tak Kim and Joo-Hyun Nam Abstract Background: We aimed to evaluate the efficacy and the safety of uterine preservation in patients with early-stage uterine adenosarcoma who want to preserve future fertility. Methods: We performed a retrospective review of patients with stage I uterine adenosarcoma diagnosed and treated at a single institute from 1998 through 2014. Results: Among the total of 31 patients, uterine preservation surgery was performed in 7 of the nulliparas. Of the 7 patients receiving uterine preservation surgery, 3 showed no evidence of disease (NED), 2 had persistent disease confined to the uterus, and 2 were alive with disease (AWD) after recurrence. One patient with an NED status had a vaginal delivery at term. In the uterine preservation group, 1 patient had sarcomatous overgrowth at the time of diagnosis and experienced disease recurrence. In the hysterectomy group, 3 of 24 patients had tumor recurrence. Of the five patients with tumor recurrence, four (80%) had sarcomatous overgrowth at diagnosis and it was significantly associated with recurrence by univariate analysis (OR 13.3, p = 0.027). Conclusions: Uterine preservation represents a possible treatment option for young female patients who want to maintain fertility. However, a detailed explanation of the risk of recurrence is necessary, especially in patients with sarcomatous overgrowth, which seems to be associated with a higher risk of recurrence. Trial registration: Retrospectively registered. Keywords: Fertility preservation, Mullerian adenosarcoma of the uterus, Uterine adenosarcoma Background a fatal outcome [3, 4]. The presence of heterologous ele- Mullerian adenosarcoma is a rare malignancy composed ments, tumor grade, and myometrial invasion depth are of benign epithelial and malignant stromal components other risk factors for metastasis [5]. and usually arises from the uterus [1]. Uterine adenosar- Due to its rarity, there are limited data on the optimal coma is considered to be a less aggressive disease than therapy (i.e., primary surgery or adjuvant therapy) for carcinosarcoma because its malignant component is uterine adenosarcoma. Total hysterectomy is generally usually low grade, and often a hysterectomy is curative considered the primary intervention for this disease, but [2]. The presence of sarcomatous overgrowth seems to uterine preservation is often desired in reproductive-age be the factor most strongly associated with an aggressive women. We aimed to analyze clinical outcomes accord- clinical course, postoperative recurrence, metastasis, and ing to which therapeutic methods were used in patients with early-stage uterine adenosarcoma to evaluate the * Correspondence: [email protected] efficacy of uterine preservation in patients wanting to Department of Obstetrics and Gynecology, University of Ulsan College of preserve their future fertility. Medicine, Asan Medical Center, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 138-736, South Korea © The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Lee et al. World Journal of Surgical Oncology (2017) 15:87 Page 2 of 5 Table 1 Characteristics of the 31 study patients with early-stage staging procedure. Sarcomatous overgrowth was de- uterine adenosarcoma fined as the presence of pure sarcoma occupying at Variables No. of patients (%) least 25% of the tumor without a benign glandular Age at diagnosis, median (range) 44.5 (21–81 years) component. We obtained Institutional Review Board Largest tumor diameter, mean (range) 4.66 (0.8–22.0 cm) approval for these studies. The relationships between variable characteristics and recurrence were assessed FIGO 2009 stage by univariate analysis using Chi-square and Fisher’s IA 21 (67.7) exact tests and by multivariate analysis using logistic IB 8 (25.8) regression analysis to identify independent risk factors IC 2 (6.5) for recurrence. Recurrence distributions were assessed Sarcomatous overgrowth by the method of Kaplan and Meier. Descriptive sta- Present 10 (32.3) tistics and data analysis were performed using SPSS ver.20.0 (SPSS Inc, Chicago, IL, USA). Absent 21 (67.7) Primary therapy Results No hysterectomy 7 (22.6) From 1998 to 2014, 33 patients were diagnosed with Hysterectomy 24 (77.4) Mullerian adenosarcoma at our institution, with 31 Adjuvant therapy meeting the inclusion criteria for our current study. One Chemotherapy 5 (16.1) of the excluded patients had stage IV disease, and the other had ovarian adenosarcoma. The characteristics of Radiotherapy 1 (3.2) the 31 study patients with early-stage uterine adenosar- Hormone therapy 1 (3.2) coma are summarized in Table 2. The median age at Total 31 (100%) diagnosis was 44.5 years, and the mean of the largest tumor diameter was 4.66 cm. Ten patients had adeno- Methods sarcoma with sarcomatous overgrowth. Thirteen patients We performed a retrospective review of patients with were under 40 years old at the time of diagnosis, and 9 stage I uterine adenosarcoma diagnosed and treated of these were nulliparas. Uterine preservation surgery, at Asan Medical Center, Seoul, Korea, from 1998 to such as hysteroscopic mass excision, cervical mass exci- 2014. Medical records were reviewed for age at diag- sion, or dilatation and curettage, was performed in 7 of nosis, largest tumor diameter, parity at diagnosis and the nulliparas. Laparoscopy-assisted vaginal hysterec- last follow-up date, primary treatment, adjuvant ther- tomy or total abdominal hysterectomy was performed in apy, recurrence, sarcomatous overgrowth, and disease 13 and 11 of the patients, respectively. Thirteen patients status at last follow-up. All cases were re-staged ac- had a bilateral salpingo-oophorectomy (BSO) at the cording to International Federation of Gynecology time of hysterectomy. Eleven patients underwent pelvic and Obstetrics (FIGO) 2009 criteria (Table 1) [6], and lymphadenectomy, while 3 patients underwent para-aortic only patients with stage I and uterine origin adeno- lymphadenectomy. Lymph node metastasis was not iden- sarcoma were included. Some patients were assigned tified in any patients. Five patients received adjuvant a clinical stage based on imaging reports and path- chemotherapy, 3 of them receiving ifosphamide with ology because they did not undergo a complete cisplatin and 2 receiving doxorubicin with cisplatin. Table 2 Clinical outcomes of the study patients with early-stage uterine adenosarcoma who received uterine preservation therapy Number Age at Stage Marital Primary therapy SO Adjuvant Recurrence Time to recurrence, Disease F/U Event diagnosis statusa therapy months status months 1 33 IB Single HSC-mass excision N No No NED 12 2 33 IA Single HSC-mass excision N MPA 3 months No NED 77 Vaginal delivery 3 40 IA Single D/C/Bx N No No NED 32 4 21 IB Single HSC-mass excision N No No Persistent 17 5 22 IA Single Cx mass excision N No No Persistent 38 6 27 IA Single HSC-mass excision Y I/P#4 Yes 13 AWD 22 TAH c BSO d/t seeding 7 27 IB Single HSC-mass excision N No Yes 27 AWD 38 aStatus at diagnosis SO sarcomatous overgrowth, F/U follow-up, HSC hysteroscopic, MPA medroxy progesterone acetate, D/C/Bx dilatation and curettage biopsy, Cx cervix, I/P ifosphamide/ cisplatin, TAH c BSO total abdominal hysterectomy with bilateral salpingo-oophorectomy, d/t due to, NED no evidence of disease, AWD alive with disease Lee et al. World Journal of Surgical Oncology (2017) 15:87 Page 3 of 5 Table 3 FIGO criteria (2009) for Mullerian adenosarcomas She was the only nullipara with sarcomatous overgrowth Stage Definition at the time of diagnosis. She underwent total abdominal I Tumor limited to uterus hysterectomy with BSO, pelvic lymph node dissection, IA Tumor limited to endometrium and endocervix with no lower anterior resection, and mass excision. myometrial invasion In the hysterectomy group, 3 of 24 patients had tumor IB Less than or equal to half myometrial invasion recurrence. One of these patients was a nullipara with sarcomatous overgrowth who received a laparoscopic IC More than half myometrial invasion hysterectomy with bilateral salpingectomy and pelvic II Tumor extends beyond the uterus, within the pelvis lymphadenectomy at another hospital. However, she had IIA Adnexal involvement disease recurrence after 5 months and returned to our IIB Tumor extends to extrauterine pelvic tissue hospital. She received pelvic mass excision with total III Tumor invades abdominal tissues (not just protruding omentectomy and chemotherapy for 6 cycles (ifospha- into the abdomen) mide and cisplatin). Another 2 patients in the hysterec- IIIA One site tomy group with tumor recurrence had sarcomatous IIIB More than one site overgrowth at diagnosis and recurred at 18 and 48 months IIIC Metastasis to pelvic and or para-aortic lymph nodes after hysterectomy, respectively. They could not receive chemotherapy due to old age and general prostration. In IV our total adenosarcoma patient series of 31 cases, 5 IVA Tumor invades bladder and or rectum (16.1%) had tumor recurrence. Four of these 5 (80%) had IVB Distant metastasis sarcomatous overgrowth at diagnosis. In the non- sarcomatous overgrowth group, 1 of 21 patients (4.8%) Adjuvant radiation was performed in 1 patient, while hor- had tumor recurrence. In the sarcomatous overgrowth mone therapy was performed in 1 other patient.
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